Original Article

Results of a bone splint technique for the treatment

of lower limb deformities in children with type I
osteogenesis imperfecta
Dasheng Lin, Wenliang Zhai, Kejian Lian, Zhenqi Ding

Abstract
Background: Children with osteogenesis imperfecta (OI) can suffer from frequent fractures and limb deformities, resulting in
impaired ambulation. Osteopenia and thin cortices complicate orthopedic treatment in this group. This study evaluates the clinical
results of a bone splint technique for the treatment of lower limb deformities in children with type I OI. The technique consists of
internal plating combined with cortical strut allograft fixation.
Materials and Methods: We prospectively followed nine children (five boys, four girls) with lower limb deformities due to type
I OI, who had been treated with the bone splint technique (11 femurs, four tibias) between 2003 and 2006. The fracture healing
time, deformity improvement, ambulation ability and complications were recorded to evaluate treatment effects.
Results: At the time of surgery the average age in our study was 7.7 years (range 5-12 years). The average length of followup was
69 months (range 6084 months). All patients had good fracture healing with an average healing time of 14 weeks (range 1216
weeks) and none experienced further fractures, deformity, or nonunion. The fixation remained stable throughout the procedure in
all cases, with no evidence of loosening or breakage of screws and the deformity and mobility significantly improved after surgery.
Of the two children confined to bed before surgery, one was able to walk on crutches and the other needed a wheelchair. The
other seven patients could walk without walking aids or support like crutches.
Conclusions: These findings suggest that the bone splint technique provides good mechanical support and increases the bone
mass. It is an effective treatment for children with OI and lower limb deformities.
Key words: Cortical strut allograft, internal fixation, osteogenesis imperfecta

Introduction

abnormal spinal curvatures.2 Children with OI can suffer

from frequent fractures and limb deformities, resulting in
impaired ambulation. Their osteopenia and thin cortices
complicate orthopedic treatment. Treatment consisting of
plate fixation alone results in high rates of refracture at the
ends of the plate due to stresses.3 Intramedullary fixation is
also associated with bone fractures, migration of implants,
penetration into the joint and hardware failure.46 Although
the cortical strut allograft has been widely used to provide
mechanical support and supplement bone healing in
revision hip arthroplasty and osseous defects,711 its use in
the treatment of lower limb deformities resulting from OI,
in children, has not been reported in literature.

steogenesis imperfecta (OI) is a genetically

heterogeneous bone disorder caused by defects
in the structure and function of type I collagen.1
OI is clinically characterized by brittle bones, osteopenia,
defective dentition, blue sclerae, loose joints and
Department of Orthopedic Surgery, The Affiliated Southeast Hospital
of Xiamen University, Orthopedic Center of Peoples Liberation Army,
Zhangzhou - 363000, China.
Address for correspondence: Dr. Zhenqi Ding,
Department of Orthopedic Surgery, The Affiliated Southeast Hospital
of Xiamen University, Orthopedic Center of Peoples Liberation Army,
Zhangzhou - 363000, China.
Email: mgss17@163.com

This study evaluates clinical results of a bone splint

technique consisting of a combined fixation of internal
plating and cortical strut allograft, for the management of
lower limb deformities in children with type I OI.

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Materials and Methods

DOI:
10.4103/0019-5413.114922

Nine children (five boys, four girls) with lower limb

377

Indian Journal of Orthopaedics | July 2013 | Vol. 47 | Issue 4

Lin, etal.: Treatment of lower limb deformities in typeI osteogenesis imperfecta

deformities due to type I OI were treated at our center using
the bone splint technique between 2003 and 2006. There
were eleven femurs and four tibias. The Ethics Committee
of our institution approved this study and parents or legal
guardians gave informed consent before surgery.

screws were put on each end of the plate. Then a cortical

strut allograft (range 918 cm) was placed opposite the
plate to span the weak part of the shaft. The cortical strut
was normally twothirds the length of the plate. The plate
and the strut graft are stabilized with screws [Figure 1].

The average age of the children was 7.7 years (range 5-12
years). All children had type I OI, based on the Sillence
classification for OI.12 All the patients had multiple fractures
before surgery (average 3.9 times, range 2-6 times). We
used a method to measure the magnitude of limb deformity.
The lines represent the axes of the proximal and distal parts
of the bone on anteroposterior and lateral radiographs and
the larger angle is the magnitude of the limb deformity.
The femoral deformity in these patients ranged from 50
to 70 (average 59) and the tibial deformity from 17 to
25 (average 21). Three children were confined to the bed
or wheelchair, four were confined indoors and two were
confined to community ambulation. Two children had a
family history of OI [Table 1].

The surgery for the tibia was similar to that for the femur.
With the aid of the Carm and tourniquet, the site of the
tibial deformity was approached using an anterolateral
incision. Wedge osteotomies were performed until the
deformity was corrected. The plate and the strut graft were
placed and stabilized with screws [Figure 2].
No external fixation was used. The patients were encouraged
to perform gentle and protective rangeofmotion exercises
from the first day after surgery. The patients, without any
other problems, were discharged one week after surgery
and were called back for followup six weeks, three months,
six months, one year and two years after surgery. The
patients were rapidly mobilized and were instructed to use
toetouch weight bearing with crutches or a walker from

Preoperative physical examination was done and

radiographs taken to precisely measure the shaft length and
the degree of deformity and to map out the exact size of
the bone wedge to be removed. The operation was done
under an epidural anesthesia or general anesthesia. The
patient was positioned on the fracture table to allow use
of a Carm machine. The strut allografts were made with
freezedried allogeneic long bones (Xinkangchen Medicine
Development, Beijing, China).

Operative procedure

The site of the femoral deformity was approached using

an anterolateral incision. The failed implant (if any) was
removed. Wedge osteotomies were performed to correct
deformity. Coxa vara and limb torsion (if present) were
corrected together. The bone of an OI patient often bends
anterolaterally. In such a case the shaft needs to be cut
completely, not leaving the contralateral cortex, but leaving
the periosteum intact where possible, to preserve the blood
supply to the bone. A plate is placed on the lateral side of
the femur to span the whole extent of the deformity. Two

Figure 1: X-ray (R) thigh with hip joint anteroposterior view showing
(a) A 12yearold boy with type I OI who had four fractures before he
was treated with the bone splint technique. Radiography showed a 65
bending angle of the femur and loosening of screws. (b) Correction of
the femoral deformity with the bone splint technique. (c) Good healing
of the fracture and bony union between the cortical strut allograft and
the host bone, two years after surgery

Table 1: Clinical details of the patients

Patient
1
2
3
4
5
6
7
8
9

Age
6
9
6
5
9
12
7
7
8

Sex
F
M
F
M
F
M
M
F
M

Limb with deformity

One femur
One femur+one tibia
One tibia
Two femurs
Two femurs
One femur
One tibia
Two femurs+one tibia
Two femurs

Indian Journal of Orthopaedics | July 2013 | Vol. 47 | Issue 4

Previous fractures
2
4+2
4
2+1
1+3
4
2
(3+1) + 2
2+2
378

Bending angle ()
58
54+19
25
68+62
50+56
65
17
(70+52) + 23
55+61

Followup (mo)
69
84
63
72
75
60
72
66
60

Healing time (w)

13
14+16
15
12+12
13+14
16
15
(15+15) + 15
12+12

Lin, etal.: Treatment of lower limb deformities in typeI osteogenesis imperfecta

deformities due to type I OI were treated at our center using
the bone splint technique between 2003 and 2006. There
were eleven femurs and four tibias. The Ethics Committee
of our institution approved this study and parents or legal
guardians gave informed consent before surgery.

screws were put on each end of the plate. Then a cortical

strut allograft (range 918 cm) was placed opposite the
plate to span the weak part of the shaft. The cortical strut
was normally twothirds the length of the plate. The plate
and the strut graft are stabilized with screws [Figure 1].

The average age of the children was 7.7 years (range 5-12
years). All children had type I OI, based on the Sillence
classification for OI.12 All the patients had multiple fractures
before surgery (average 3.9 times, range 2-6 times). We
used a method to measure the magnitude of limb deformity.
The lines represent the axes of the proximal and distal parts
of the bone on anteroposterior and lateral radiographs and
the larger angle is the magnitude of the limb deformity.
The femoral deformity in these patients ranged from 50
to 70 (average 59) and the tibial deformity from 17 to
25 (average 21). Three children were confined to the bed
or wheelchair, four were confined indoors and two were
confined to community ambulation. Two children had a
family history of OI [Table 1].

The surgery for the tibia was similar to that for the femur.
With the aid of the Carm and tourniquet, the site of the
tibial deformity was approached using an anterolateral
incision. Wedge osteotomies were performed until the
deformity was corrected. The plate and the strut graft were
placed and stabilized with screws [Figure 2].
No external fixation was used. The patients were encouraged
to perform gentle and protective rangeofmotion exercises
from the first day after surgery. The patients, without any
other problems, were discharged one week after surgery
and were called back for followup six weeks, three months,
six months, one year and two years after surgery. The
patients were rapidly mobilized and were instructed to use
toetouch weight bearing with crutches or a walker from

Preoperative physical examination was done and

radiographs taken to precisely measure the shaft length and
the degree of deformity and to map out the exact size of
the bone wedge to be removed. The operation was done
under an epidural anesthesia or general anesthesia. The
patient was positioned on the fracture table to allow use
of a Carm machine. The strut allografts were made with
freezedried allogeneic long bones (Xinkangchen Medicine
Development, Beijing, China).

Operative procedure

The site of the femoral deformity was approached using

an anterolateral incision. The failed implant (if any) was
removed. Wedge osteotomies were performed to correct
deformity. Coxa vara and limb torsion (if present) were
corrected together. The bone of an OI patient often bends
anterolaterally. In such a case the shaft needs to be cut
completely, not leaving the contralateral cortex, but leaving
the periosteum intact where possible, to preserve the blood
supply to the bone. A plate is placed on the lateral side of
the femur to span the whole extent of the deformity. Two

Figure 1: X-ray (R) thigh with hip joint anteroposterior view showing
(a) A 12yearold boy with type I OI who had four fractures before he
was treated with the bone splint technique. Radiography showed a 65
bending angle of the femur and loosening of screws. (b) Correction of
the femoral deformity with the bone splint technique. (c) Good healing
of the fracture and bony union between the cortical strut allograft and
the host bone, two years after surgery

Table 1: Clinical details of the patients

Patient
1
2
3
4
5
6
7
8
9

Age
6
9
6
5
9
12
7
7
8

Sex
F
M
F
M
F
M
M
F
M

Limb with deformity

One femur
One femur+one tibia
One tibia
Two femurs
Two femurs
One femur
One tibia
Two femurs+one tibia
Two femurs

Indian Journal of Orthopaedics | July 2013 | Vol. 47 | Issue 4

Previous fractures
2
4+2
4
2+1
1+3
4
2
(3+1) + 2
2+2
378

Bending angle ()
58
54+19
25
68+62
50+56
65
17
(70+52) + 23
55+61

Followup (mo)
69
84
63
72
75
60
72
66
60

Healing time (w)

13
14+16
15
12+12
13+14
16
15
(15+15) + 15
12+12

Lin, etal.: Treatment of lower limb deformities in typeI osteogenesis imperfecta

one week after surgery. The patients progressed to full
weight bearing when they had the strength and balance
to do so. They were instructed to leave the crutch support
when they were able to walk without a substantial limp.
Cyclical intravenous pamidronate administration was
given to all patients on three consecutive days, every
four months. The dose was one milligram per kilogram
of body weight per day. The dose was reduced by half
during the first day of pamidronate treatment, then full
doses 2nd and 3rd day.13

stable throughout the followup in all cases, with no evidence

of loosening or breakage of screws.
One year postsurgery, the allografts were incorporated
into the host bones. The medullary cavity junction
between the allograft and the host bone was recognized
[Figures 1 and 2]. After surgery, the deformity and mobility
significantly improved (P < 0.05, Table 2). Of the two
children confined to bed before surgery, one was able to
walk on crutches and the other needed a wheelchair. The
other seven patients could walk without support.

Postoperative evaluation

Discussion

The clinical and radiological outcomes of all subjects were

evaluated.14 The clinical evaluation covered healing of
bone fractures, improvement in deformities, incidence
of infection and degree of ambulation (i.e., confined to
bed, wheelchair, indoors, community, or independently
walking). The radiological evaluation included the callus
gray density, position, migration, bending and breakage
of internal fixation and refracture rates.

Osteopenia and local deformities are common in children

with OI and often associated with multiple fractures.
Reposition and plaster cast fixation can result in satisfactory
results temporarily. However, long term immobilization
results in further reduction in bone mass and poor bone
stock, thereby increasing the risk of further fractures and
worsening deformities. Thus, a vicious cycle of fracture,
immobilization and refracture set in.15

Results

Saldanha et al.16 have reported the use of external fixation

to correct limb deformity. However, children with OI often
have associated osteopenia and thin cortices and fixator
screws do not have enough purchase in such bones. Implant
loosening, fracture displacement or more severely, recurrent
fractures frequently occur in these cases. Similar problems
exist when internal bone plating is used alone. In addition,
the stress shielding effect associated with bone plating
further leads to reduction in bone density, aggravating the
osteopenia.3

All the patients had a followup period of at least five years

and the mean followup was 69 months (range 6084
months). All the surgical wounds healed with primary
intention. Allograft rejection, neurovascular complications
or infections did not occur in any patient. All the patients
healed well, with an average healing time of 14 weeks
(range 1216 weeks) and the callus gray density increased
with the passing of time. None of the patients had
refractures, deformity or nonunion. The fixation remained

All the patients in our study had undergone unsuccessful

orthopedic operations before they had been referred to our
institution. Four had been treated with plating (four femurs,
two tibias) and two with external fixation (three femurs).
Refractures occurred shortly after these unsuccessful
operations and the deformities were aggravated.
The mainstay of treatment for deformity in the lower limb
caused by OI are multiple osteotomies and intramedullary
nailing, which can correct the deformity, increase limb
strength, reduce the risk of further fractures and increase limb
length. There is wide variability in the types of intramedullary
a

Table 2: Preoperative and postoperative walking ability of

patients

Figure 2: X-ray (L) leg bones anteroposterior view showing (a) A

sixyearold girl with type I OI who had experienced four fractures
before she was treated with the bone splint technique. Radiography
showed a 25 bending angle of the tibia, aggravation of the deformity
and loosening of screws before surgery. (b) Correction of the tibial
deformity with the bone splint technique. (c) The bony union between
the cortical strut allograft and the host bone at two years after surgery

No. of
patients

Confined Indoors Community Independently

to bed or
walking
wheelchair
Preoperative
3
4
2
0
Postoperative
1
0
1
7
379

Indian Journal of Orthopaedics | July 2013 | Vol. 47 | Issue 4

Lin, etal.: Treatment of lower limb deformities in typeI osteogenesis imperfecta

rods and their clinical results.46,1720 With bone growth, the rod
becomes relatively shorter and require revisions when the
patient outgrows the rod. The complications of intramedullary
fixation include fractures, migration of implants, joint
penetration and hardware loosening or disengagement.

biomechanical strength is improved. In conclusion, this

bone splint technique is an effective treatment for lower
limb deformities in children with type I OI.

In our study, lower limb deformities in children caused by

type I OI were treated with osteotomies and internal plating,
combined with cortical strut allografts. This technique
effectively rebuilt the thin cortex and helped the screws
gain better purchase on the bone, thus preventing the
fixation from loosening.21 The screws were inserted through
three layers of cortices to achieve a socalled bone splint
technique composed of the steel plate, screws and allograft.
The bone splint technique shared the load with the bone
and decreased the bending and torsion stress on the plate.
As a result, the entire mechanical performance of the
fixation was increased and the risks of fracture at the ends of
the implant or internal fixation failures were reduced. Due to
the stable fixation, the creeping substitution of the allograft
and bone healing proceeded smoothly. After the union
occurred, the allograft underwent adaptive remodeling
secondary to physiological load bearing.22 In addition, the
bone splint technique blocked the cartilage from intruding
into the fracture section, favoring the healing of the bone
fracture. In our study, during the 69month followup, no
patient had failure of internal fixation. Radiography showed
that 12-16 weeks after surgery, the bone healed well. One
year after surgery, the allograft was mostly incorporated
into the host bone. The bony union of the allograft and the
host bone greatly increased the bone mass and improved
the strength of the bone.

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References

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The bone splint technique described herein can provide
additional support to the host bone, decrease stress on
the implant and reduce the risks of recurring fractures
or fixation failures. After bony union of the allograft and
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Indian Journal of Orthopaedics | July 2013 | Vol. 47 | Issue 4

380

Lin, etal.: Treatment of lower limb deformities in typeI osteogenesis imperfecta

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How to cite this article: Lin D, Zhai W, Lian K, Ding Z. Results of
a bone splint technique for the treatment of lower limb deformities
in children with type I osteogenesis imperfecta. Indian J Orthop
2013;47:377-81.
Source of Support: This study was supported by the key projects
from Nanjing Military Region during the 11th Five Year Plan Period
(No. 06MA97)., Conflict of Interest: None.

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